Intra-abdominal telangiectatic osteosarcoma: a case report.

BACKGROUND: Telangiectatic osteosarcoma is rare and it rarely affects flat bones, especially the bones of the pelvis. It is uncommon for telangiectatic osteosarcoma to be considered as a differential diagnosis when assessing a large intrabdominal mass. CASE REPORT: We present our case of a 33-year-old African female who presented with a sizeable telangiectatic osteosarcoma of the left iliac bone. She reported a 3-year duration of a painless, slow-growing mass arising from the left flank. At examination, a large bony hard mass extending from the left ilium to the umbilicus was noted, almost mimicking an intra-abdominal pregnancy. All laboratory tests were within normal limits and an unconventional surgical approach was used for a one-stage excision of the tumor without complications. The definitive histopathological diagnosis postexcision was that of a telangiectatic osteosarcoma only on the second review of the histological specimen. CONCLUSIONS: Pelvic telangiectatic osteosarcoma is rare, and the ilium is the commonly affected pelvic bone. These tumors can be sizeable at presentation with intra-abdominal or pelvic extension with a high chance of misdiagnosis. Fortunately surrounding soft tissue involvement seems to be a rare and late finding when present.

The effect of preoperative embolization on giant cell tumors of the bone localized in the iliosacral region of the pelvis.

INTRODUCTION: Giant cell tumors of the bone (GCTB) are aggressive neoplasms, with rare occurrences in the posterior pelvis and sacral area. Surgical challenges in this region include the inability to apply a tourniquet and limited cementation post-curettage due to proximity to neurovascular structures, leading to potential complications. This case-control study explores the impact of preoperative embolization on GCTB located in the iliosacral region. METHODS: Five surgeries (January-December 2021) for pelvic GCTB (3 sacrum, 2 posterior ilium) were performed on four patients. Diagnosis was confirmed through preoperative CT-guided biopsies. One surgery involved curettage with PMMA cement filling, while four surgeries had curettage without cavity filling. Preoperative embolization of the tumor feeding vessel occurred approximately 16 h before surgery in two cases. Denosumab treatment was not administered. RESULTS: Tumor volume, assessed by preoperative MRI, was comparable between patients with and without preoperative embolization (p = .14). Surgeries without embolization had a mean intraoperative blood loss of 3250 ml, erythrocyte transfusion volume of 1125 ml, and a mean surgical time of 114.5 min for two surgeries. Surgeries with preoperative embolization showed a mean intraoperative blood loss of 1850 ml, no erythrocyte transfusion requirement, and a mean surgical time of 68 min. CONCLUSION: Curettage of GCTB in the posterior pelvis and sacrum presents challenges, with significant intraoperative blood loss impacting surgical time and transfusion needs. Preoperative embolization may be beneficial in reducing blood loss during surgery in these cases.

Approaching virtual osteoid volume estimation and in-depth tissue characterization in patients with tumor-induced osteomalacia.

Tumor-induced osteomalacia (TIO) poses a significant diagnostic challenge, leading to increased disease duration and patient burden also by missing clinical suspicion. Today, diagnosis of osteomalacia relies on invasive iliac crest biopsy, if needed. Therefore, a noninvasive method would be beneficial for patients with severe osteomalacia, such as TIO, to inform their clinical management and address specific needs, like estimating the regeneration capacity at high osteoid volumes (OVs) or the potential of a hungry bone syndrome after tumor removal. Furthermore, given the lack of comprehensive histological characterization of TIO, there is a need for additional tissue characterization. Therefore, our assessment encompassed iliac crest biopsies that were examined using quantitative electron backscattered microscopy, Raman spectroscopy, micro-computed tomography, and histology to analyze the biopsy tissue. Our clinical assessment encompassed DXA and high-resolution peripheral quantitative computed tomography (HR-pQCT) alongside with biochemical analyses and clinical evaluations. Combining imaging and clinical data, we established a model to predict the OV. We compared 9 TIO patients with 10 osteoporosis (OPO) patients and 10 healthy controls. Histological analyses confirmed a pronounced OV in TIO patients (OPO: 1.20% ± 1.23% vs TIO: 23.55% ± 12.23%, P < .0005), and spectroscopy revealed lower phosphate levels in TIO biopsies. By combining HR-pQCT and laboratory diagnostics, we developed a linear regression model to noninvasively predict the OV revealing significantly higher modeled OV/BVmodel values of 24.46% ± 14.22% for TIO compared to the control group (5.952% ± 3.44%, P ≤ .001). By combining laboratory diagnostics, namely, ALP and Tt.BMDRadius measured by HR-pQCT, we achieved the calculation of the virtual osteoid volume to bone volume ratio (OV/BVmodel) with a significant correlation to histology as well as reliable identification of TIO patients compared to OPO and control. This novel approach is potentially helpful for predicting OV by noninvasive techniques in diagnostic procedures and improving the clinical management of TIO.

Osteomalacia, a bone mineralization disease, results in soft bones due to a lack of calcium or phosphate. Tumor-induced osteomalacia (TIO) is an acquired and challenging form of osteomalacia due to low serum phosphate levels that often lead to prolonged patient suffering. Current diagnosis of osteomalacia involves surgical bone biopsies, but a noninvasive approach would be beneficial, improving clinical management and addressing specific needs like estimating the bone's quality and ability to recover. We used advanced techniques like electron microscopy, spectroscopy, and high-resolution CT to study bone samples from 9 TIO patients. Additionally, we assessed their bone health through sophisticated imaging and blood analyses. Microscopy confirmed huge amounts of soft bone tissue due to a severe mineralization defect. By combining imaging and blood analysis, we developed a noninvasive method to predict the amount of soft tissue (osteoid) to understand soft bones without the need for surgical interventions. In conclusion, our innovative approach, combining blood diagnostics (alkaline phosphatase) with total BMD from high-resolution 3D clinical imaging of the lower arm, allows us to predict the osteoid amount virtually. This method can also compare TIO patients with controls or those with osteoporosis and might be helpful in the future.

Hemangioma of the Ilium Simulating Bone Metastasis on 18 F-PSMA-1007 PET/CT.

ABSTRACT: A 64-year-old man was referred for 18 F-PSMA-1007 PET/CT scan for initial staging of biopsy-proved prostate adenocarcinoma. 18 F-PSMA-1007 PET/CT showed focal intense 18 F-PSMA-1007 of the prostate adenocarcinoma and a focal intense activity with SUV max of 10.5 in the left ilium. The 18 F-PSMA-1007-avid iliac bone lesion corresponded to a hemangioma, which was initially detected on pelvic MRI 13 months ago and remained stable in size. This case indicates that hemangioma should be included in the differential diagnosis of PSMA-avid iliac bone lesions.

[A Case of Prostate Cancer Diagnosed by the Bone Biopsy of the Right Iliac Metastatic Lesion].

A 73-year-old man was referred to our hospital because of a high prostate specific antigen (PSA) level. The PSA level at our hospital was 63.5 ng/ml. Pelvic magnetic resonance imaging (MRI) showed findings strongly suggestive of multiple pelvic bone metastases, but no obvious malignant findings in the prostate. A 12-core prostate biopsy was performed and no cancer was detected. Computed tomography and bone scintigraphy showed findings suspicious of bone metastases in the sternum, thoracolumbar spine, pelvic bone, and sacrum. Spine MRI revealed a mass in the vertebral body from the eighth thoracic vertebra to the first lumbar vertebra. A biopsy of the right iliac crest showed adenocarcinoma and was positive for PSA staining, leading to the diagnosis of multiple bone metastases of prostate cancer. Abiraterone acetate in combination with androgen deprivation was started. He received medication and radiation therapy to his sternum for pain relief. Spine MRI after 4 months showed decreased vertebral body weights and serum PSA levels were ＜0.003 ng/ml after 5 months. Seventeen months after treatment, PSA remains below 0.003 ng/ml, and the patient is currently pain-free.

Bone intrinsic material and compositional properties in postmenopausal women diagnosed with long-term Type-1 diabetes.

The incidence of diabetes mellitus and the associated complications are growing worldwide, affecting the patients' quality of life and exerting a considerable burden on health systems. Yet, the increase in fracture risk in type 1 diabetes (T1D) patients is not fully captured by bone mineral density (BMD), leading to the hypothesis that alterations in bone quality are responsible for the increased risk. Material/compositional properties are important aspects of bone quality, yet information on human bone material/compositional properties in T1D is rather sparse. The purpose of the present study is to measure both the intrinsic material behaviour by nanoindentation, and material compositional properties by Raman spectroscopy as a function of tissue age and microanatomical location (cement lines) in bone tissue from iliac crest biopsies from postmenopausal women diagnosed with long-term T1D (N = 8), and appropriate sex-, age-, BMD- and clinically-matched controls (postmenopausal women; N = 5). The results suggest elevation of advanced glycation endproducts (AGE) content in the T1D and show significant differences in mineral maturity / crystallinity (MMC) and glycosaminoglycan (GAG) content between the T1D and control groups. Furthermore, both hardness and modulus by nanoindentation are greater in T1D. These data suggest a significant deterioration of material strength properties (toughness) and compositional properties in T1D compared with controls.

Osteomalacia Prevalence, Biochemical Profile, and Histology in Patients with Low-Energy Hip Fractures Over the Age of 45.

Our objective was to determine the prevalence of osteomalacia in low-energy hip fracture patients over the age of 45, based on biochemical and histological measures. This cross-sectional study included 72 patients over 45 with low-energy mechanism hip fractures. Samples of fasting venous blood were taken for hemograms and serum biochemistry analyses. Bicortical biopsies of the iliac crest were obtained, processed, and evaluated by an expert pathologist for osteomalacia. Biochemical osteomalacia (b-OM) is defined according to a distinct criterion. A low level of serum calcium, phosphorus, albumin, and 25OHD was found in 43.1, 16.7, 73.6, and 59.7% of patients, respectively. 50.0% of patients had high serum alkaline phosphatase (ALP) levels. b-OM was found in 30 (41.7%), and no significant association was found with PTH, Cr, Alb, age, sex, fracture type, side of the trauma, and season were not associated with osteomalacia. Osteomalacia was diagnosed on histopathological analysis in 19/72 (26.7%), and 54/72 (75.0%) of all cases fulfilled b-OM criteria. In the histologic evaluation, osteoid seam width, osteoid surface, and osteoid volume were 28.5 µm, 25.6, and 12.1%, respectively. The sensitivity, specificity, positive predictive value, negative predictive value, and accuracy of the biochemical test for detecting osteomalacia were 73.6, 64.2, 42.4, 87.2, and 66.7%, respectively. Up to 30% of elderly patients with low-energy hip fractures are affected by osteomalacia. A biochemical screening along with a bone biopsy and histopathologic evaluation may be logical in a high-risk population for osteomalacia diagnosis.

(Smoldering) multiple myeloma: mismatch between tumor load estimated from bone marrow biopsy at iliac crest and tumor load shown by MRI.

In multiple myeloma and its precursor stages, precise quantification of tumor load is of high importance for diagnosis, risk assessment, and therapy response evaluation. Both whole-body MRI, which allows to investigate the complete bone marrow of a patient, and bone marrow biopsy, which is commonly used to assess the histologic and genetic status, are relevant methods for tumor load assessment in multiple myeloma. We report on a series of striking mismatches between the plasma cell infiltration estimating the tumor load from unguided biopsies of the bone marrow at the posterior iliac crest and the tumor load assessment from whole-body MRI.

Inter and intra-observer agreement of the 3-dimensional CT based anterior inferior iliac spine classification system shows fair-to-moderate agreement among high volume hip surgeons.

PURPOSE: The purpose of this multicenter, multinational study is to evaluate the agreement level of the Hetsroni's classification system across high-volume hip surgeons who specialize in hip preservation surgeries. METHODS: Four surgeons from three countries reviewed a digital survey that included 93 3D CT images of the hip from 53 patients. The population was composed of individuals who had undergone a pelvis CT scan in a tertiary hospital between 2000 and 2016. Each rater reviewed the images and classified each image according to AIIS subtype I, II, or III. After a minimum of two months, the raters repeated the survey. The inter-rater and intra-rater agreement was then assessed. The kappa values were calculated to determine variability. RESULTS: Inter-rater agreement levels yielded fair agreement for both sessions (Kappa = 0.4, p value < 0.001 in the first and Kappa = 0.27, p value < 0.001 in the second). Inter-rater agreement levels separating non-pathological Type I from pathological Types II and III yielded moderate to fair inter-rater agreement levels (K = 0.47, p value < 0.001 in the first session and k = 0.32, p value < 0.001 in the second). Intra-rater reliability displayed moderate agreement (average K = 0.53). CONCLUSION: The current 3D CT-based AIIS classification system shows fair-to-moderate inter- and intra-rater agreement among high-volume hip surgeons. According to this study, the agreement of the Hetsroni classification system is not able to be sufficiently reproduced. Since accurate classification of the AIIS morphology is imperative in establishing proper treatment for SSI, this classification system there is therefore limited in its clinical value. LEVEL OF EVIDENCE: III.

Treatment of postmenopausal osteoporosis patients with teriparatide for 24 months reverts forming bone quality indices to premenopausal healthy control values.

Postmenopausal osteoporosis (PMOP) therapies are frequently evaluated by bone mineral density (BMD) gains against patients receiving placebo (calcium and vitamin D supplementation, a mild bone turnover-suppressing intervention), which is not equivalent to either healthy or treatment-naive PMOP. The aim of the present observational study was to assess the effects of TPTD treatment in PMOP (20 µg, once daily) at 6 (TPTD 6m; n = 28, age 65 ± 7.3 years), and 24 (TPTD 24m; n = 32, age 67.4 ± 6.15 years) months on bone quality indices at actively forming trabecular surfaces (with fluorescent double labels). Data from the TPTD-treated PMOP patients were compared with those in healthy adult premenopausal women (HC; n = 62, age 40.5 ± 10.6 years), and PMOP receiving placebo (PMOP-PLC; n = 94, age 70.6 ± 4.5 years). Iliac crest biopsies were analyzed by Raman microspectroscopy at three distinct tissue ages: mid-distance between the second label and the bone surface, mid-distance between the two labels, and 1 µm behind the first label. Mineral to matrix ratio (MM), mineral maturity/crystallinity (MMC), tissue water (TW), glycosaminoglycan (GAGs), and pyridinoline (Pyd) content were determined. Outcomes were compared by ANCOVA with subject age and tissue age as covariates, and health status as a fixed factor, followed by Sidak's post-hoc testing (significance assigned to p < 0.05). Both TPTD groups increased MM compared to PMOP-PLC. While TPTD 6m had values similar to HC, TPTD 24m had higher values compared to either HC or TPTD 6m. Both TPTD groups had lower MMC values compared to PMOP-PLC and similar to HC. TPTD 6m patients had higher TW content compared to HC, while TPTD 24m had values similar to HC and lower than either PMOP-PLC or TPTD 6m. Both TPTD groups had lower GAG content compared to HC group, while TPTD 6m had higher values compared to PMOP-PLC. Finally, TPTD 6m patients had higher Pyd content compared to HC and lower compared to PMOP-PLC, while TPTD 24m had lower values compared to PMOP-PLC and TPTD 6m, and similar to HC group. The results of the present study indicate that effects of TPTD on forming trabecular bone quality indices depend on treatment duration. At the recommended length of 24 m, TPTD restores bone mineral and organic matrix quality indices (MMC, TW, Pyd content) to premenopausal healthy (HC) levels.

Absceso del psoas ilíaco secundario a un tumor de colon izquierdo fistulizado al retroperitoneo: caso clínico y análisis de la literatura / Iliopsoas abscess resulting from penetrating left colon cancer to the retroperitoneum: case report and literature review / Abscesso do iliopsoas secundário a tumor de cólon esquerdo fistulizado para o retroperitônio: caso clínico e análise da literatura

Los abscesos del psoas ilíaco secundarios a un tumor de colon fistulizado son excepcionales y potencialmente graves. La mayoría son adenocarcinomas de tipo mucinoso. Su tratamiento es complejo ya que, para lograr una resección oncológica pretendidamente curativa, es necesario realizar una resección ampliada con mayor morbimortalidad. Presentamos el caso de una paciente con un adenocarcinoma mucinoso de colon izquierdo fistulizado al músculo ilíaco y la pared anterolateral del abdomen en la que se realizó una resección multivisceral que incluyó el colon izquierdo, el músculo y la cresta ilíaca y parte de la pared anterolateral del abdomen.

Iliopsoas abscess secondary to perforation of colon cancer is an extremely rare and potentially life-threatening condition. Most tumors are mucinous adenocarcinomas. Its treatment its complex, as most patients need radical extended resections to achieve good oncological results, which are in turn, graved with higher morbidity and mortality. We present the case of a patient with a left colon mucinous adenocarcinoma penetrating to the iliopsoas muscle and the anterolateral abdominal wall that required a multivisceral resection including left colon, iliac muscle and crest and part of the anterolateral abdominal wall.

Abscessos do iliopsoas secundários a um tumor de cólon fistulizado são raros e potencialmente graves. A maioria são adenocarcinomas do tipo mucinoso. Seu tratamento é complexo, pois, para se obter uma ressecção oncológica supostamente curativa, é necessário realizar uma ressecção ampliada com maior morbimortalidade. Apresentamos o caso de um paciente com adenocarcinoma mucinoso de cólon esquerdo fistulizado para o músculo ilíaco e parede ântero-lateral do abdome no qual foi realizada ressecção multivisceral que incluiu cólon esquerdo, músculo e crista ilíaca e parte do a parede anterolateral do abdome.

Diverticulitis apendicular, como diagnóstico diferencial / Appendiceal diverticulitis as differential diagnosis

RESUMEN La diverticulitis apendicular (DA) es una patología poco frecuente, considerada clínicamente indistinguible de la apendicitis aguda, aunque podría presentar una sintomatología más leve. Este es el caso de un paciente masculino de 59 años, que concurre al Servicio de Urgencias presentando signos y síntomas sugestivos de una apendicitis aguda; una ecografía informa un asa tubular parcialmente compresible de 7,8 mm de diámetro y una fina banda de líquido laminar, compatible con proceso apendicular agudo. La apendicectomía se realizó de manera convencional evidenciándose un apéndice inflamado principalmente en su región distal. La histología reveló diverticulitis apendicular complicada con rotura. El paciente evolucionó favorablemente y se externó a las 24 horas. Existe una asociación de DA y neoplasia apendicular, por lo que se recomienda una colonoscopia y el seguimiento de este tipo de pacientes.

ABSTRACT Appendiceal diverticulitis (AD) is a rare condition considered clinically identical to acute appendicitis although it may present milder symptoms. We report the case of a 59-year-old male patient who visited the emergency department due to signs and symptoms suggestive of acute appendicitis. An abdominal ultrasound showed partially compressible tubular loop with a diameter of 7.8 mm and a thin band of laminar fluid, consistent with acute appendiceal process. During conventional appendectomy the appendix had signs of inflammation, mainly in the distal region. The histology revealed appendiceal diverticulitis complicated with rupture. The patient had favorable outcome and was discharged 24 hours later. As, there is a clear association between AD and appendiceal neoplasms, colonoscopy and patient monitoring is recommended.

Bone Mineral and Organic Properties in Postmenopausal Women Treated With Denosumab for Up to 10 years.

In postmenopausal women with osteoporosis, denosumab (DMAb) therapy through 10 years resulted in significantly higher degree of mineralization of bone, with a subsequent increase from years 2-3 to year 5 and no further difference between years 5 and 10. Our aim was to assess the variables reflecting the quality of bone mineral and organic matrix (Fourier transform infrared microspectroscopy), and the microhardness of bone (Vickers microindentation). Cross-sectional assessments were performed in blinded fashion on iliac bone biopsies from osteoporotic women (72 from FREEDOM trial, 49 from FREEDOM Extension trial), separately in cortical and cancellous compartments. After 2-3 years of DMAb, mineral/matrix ratio and microhardness of cortical bone were significantly higher compared with placebo, whereas mineral maturity, mineral crystallinity, mineral carbonation, and collagen maturity were not different in both bone compartments. Through 5 years of DMAb, mineral carbonation was significantly lower and mineral/matrix ratio, mineral maturity, and crystallinity were significantly higher versus 2-3 years and were not different between 5 and 10 years, with the exception of mineral maturity in cancellous bone. These data support a transition of mineral to more mature crystals (within physiological range) and the completeness of secondary mineralization within 5 years of DMAb treatment. Microhardness in cortical and cancellous compartments was significantly lower at 5 years of DMAb versus 2-3 years and was not different from years 5 to 10. The lower microhardness at years 5 and 10 is likely the result of maturation of the organic matrix in a persistently low state of bone remodeling over 5 and 10 years. © 2022 American Society for Bone and Mineral Research (ASBMR).

Evaluation of a novel classification system to predict local recurrence in sarcoma patients undergoing hemipelvectomy with iliosacral resection.

AIMS: Iliosacral sarcoma resections have been shown to have high rates of local recurrence (LR) and poor overall survival. There is also no universal classification for the resection of pelvic sarcomas invading the sacrum. This study proposes a novel classification system and analyzes the survival and risk of recurrence, when using this system. METHODS: This is a retrospective analysis of 151 patients (with median follow-up in survivors of 44 months (interquartile range 12 to 77)) who underwent hemipelvectomy with iliosacral resection at a single centre between 2007 and 2019. The proposed classification differentiates the extent of iliosacral resection and defines types S1 to S6 (S1 resection medial and parallel to the sacroiliac joint, S2 resection through the ipsilateral sacral lateral mass to the neuroforamina, S3 resection through the ipsilateral neuroforamina, S4 resection through ipsilateral the spinal canal, and S5 and S6 contralateral sacral resections). Descriptive statistics and the chi-squared test were used for categorical variables, and the Kaplan-Meier survival analysis were performed. RESULTS: Resections were S1 in 25/151 patients (17%), S2 in 70/151 (46%), S3 in 33/151 (22%), S4 in 77/151 (11%), S5 in 4/151 (3%), and S6 in 2/151 (1%). An internal hemipelvectomy was performed in 113/151 patients (75%), and 38/151 patients (25%) had an external hemipelvectomy. The predominant types of sarcoma were high-grade osteosarcoma in 48/151 patients (32%), chondrosarcoma in 41/151 (27%), Ewing sarcoma in 33/151 (22%), pleomorphic sarcoma in 17/151 (11%), and others in 2/151 (8%). LR was found in 24/151 patients (15%) with S3, S5, with S6 resections showing the highest rate of LR (p = 0.038). Overall, 19/151 patients (16%) had evidence of metastastic disease at the time of surgery and these patients showed poorer survival when compared to patients with no metastasis. CONCLUSION: The proposed classification can help to report and compare different surgical and reconstructive approaches in these difficult cases who are still have a considerable risk of LR. Cite this article: Bone Joint J 2022;104-B(2):290-296.

Magnetic Resonance Imaging Is Not Reliable in Classifying Anterior Inferior Iliac Spine Morphology Compared to 3-Dimensional Computerized Tomography.

PURPOSE: The purpose of this study is to determine the reliability of standard magnetic resonance imaging (MRI) evaluation of AIIS morphology compared with three-dimensional (3D) computerized tomography (CT) (reference standard). METHODS: Sixty hips in 30 patients met the inclusion criteria. The images were reviewed and classified by two fellowship-trained orthopedic surgeons. A second imaging viewing session was conducted in the same manner for validation of AIIS evaluation. The agreement and accuracy indices between the two raters were calculated for each imaging modality (inter-rater agreement) as well the agreement across the imaging modality for each rater (intermethod agreement). RESULTS: The inter-rater agreement for the morphological evaluation of the AIIS for the first session according to 3D CT was .553 (P < .05) and by means of MRI was .0163 (P < .05). The inter-rater agreement for the second session by means of 3D CT was .449 (P < .05) and according to MRI was 0 (P < .05). The inter-method agreement for rater 1 for the first session was .04 (P < 0.05), while the agreement for rater 2 was .016 (P < .05). The intermethod agreement for rater 1 on the second session was .35 (P < 0.05), while that of rater 2 was .297(P < .05). The overall accuracy of MRI compared to 3D CT for rater 1 for the first session was .531, .490, and .959 for types I, II, III respectively and .551, .531, and .980 for the second session for types I, II, and III respectively. The overall accuracy of MRI compared to 3D CT for rater 2 for the first session was .551, .469, and .918 for types I, II, III respectively and .633, .592, and .918 for the second session for types I, II, and III, respectively. CONCLUSION: MRI evaluations and subsequent classifications of AIIS morphology demonstrated a poor to slight correlation compared with that of the reference standard of 3D CT. LEVEL OF EVIDENCE: Level II, retrospective diagnostic study.

Case report: Osteomalacia due to bisphosphonate treatment in a patient on hemodialysis.

BACKGROUND: No publications have reported on osteomalacia in patients receiving intermittent cyclical therapy with etidronate (a bisphosphonate) and undergoing long-term hemodialysis (HD). CASE PRESENTATION: We report on a 46-year-old Japanese man admitted to our hospital for further examination of left forearm pain. Maintenance HD was started at age 24 years, and the man had been on HD since then. At age 38 years, surgical parathyroidectomy was performed for secondary hyperparathyroidism; iliac crest bone biopsy performed at the same time showed osteitis fibrosa. The active vitamin D3 preparation calcitriol was started, and intermittent cyclical etidronate therapy was introduced 2 years later for osteoporosis. At age 45 years, the patient stopped taking calcitriol because of hypercalcemia but continued with etidronate. At age 46 years, a pseudofracture with a Looser zone occurred in the left ulna, and left femur bone biopsy revealed osteomalacia. Etidronate was discontinued, and calcitriol was restarted; open reduction and internal fixation with an angular stability plate were performed. Union of the bone was achieved 10 months after the operation. At age 49 years, a lumber bone biopsy confirmed improved bone morphometry. CONCLUSIONS: We believe that intermittent cyclical etidronate therapy without administration of active vitamin D3 during long-term HD might have induced osteomalacia, resulting in the ulna insufficiency fracture. Therefore, we propose that administration of active vitamin D3 is essential to prevent osteomalacia in patients on long-term HD who are receiving bisphosphonates and have potential vitamin D3 deficiency.

Future studies using histomorphometry in type 1 diabetes mellitus.

PURPOSE OF REVIEW: This article reviews the current state of research in type 1 diabetes and bone, focusing on human bone turnover markers and histomorphometry. RECENT FINDINGS: Bone turnover markers have been used for decades to document static bone turnover status in a variety of diseases but especially in diabetes. Two new studies focus on dynamic testing conditions to examine the acute effects of insulin and exercise on bone turnover. Publications of human bone histomorphometry in type 1 diabetes are few but there are several new studies currently underway. SUMMARY: Here, we review the most recent literature on human bone turnover markers and histomorphometry. Low bone turnover is thought to be a major underlying factor in bone fragility in T1DM. Further studies in human transilial bone biopsies will be helpful in determining the mechanisms.

An Isolated Iliac Wing Stress Fracture in a Male Marathon Runner: A Case Report.

CASE: A 49-year-old man, who had started jogging 3 months before his first participation in a marathon race, presented with acute pain and difficulty walking that began during the marathon race. Tumors and bone metabolism factors were ruled out by blood examination and various imaging findings. Isolated iliac wing fatigue fracture was diagnosed and treated with conservative therapy. CONCLUSION: This is the first English-literature report we know of an isolated iliac wing stress fracture in a male marathon runner without underlying disease. Our study highlights the importance of considering this rare fracture when diagnosing patients presenting with iliac pain.